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Suspected Trigeminal Nerve Neuropathy Causing Persistent ISSN 0008-3194 (p)/ISSN 1715-6181 (e)/2019/126–138/$2.00/©JCCA 2019 Suspected trigeminal nerve neuropathy causing persistent idiopathic facial pain: a report of four cases Nicholas Moser, BSc, DC, FCCS(C)1 Brad Muir, HBSc(Kin), DC, FRCCSS(C)2 Persistent idiopathic facial pain is often a disabling La douleur faciale idiopathique persistante est souvent condition for patients. Due to a lack of agreed upon une condition invalidante pour les patients. En raison de diagnostic criteria and varied symptomatology, the l’absence de critères diagnostiques convenus et d’une diagnosis of persistent idiopathic facial pain is elusive symptomatologie variée, le diagnostic de douleur faciale and remains one of exclusion. It is typically described as idiopathique persistante est difficile à établir et demeure a unilateral, deep, poorly localized pain in the territory un diagnostic d’exclusion. On décrit généralement of the trigeminal nerve, however there are a number of la douleur comme étant unilatérale, profonde et mal case reports that describe bilateral symptoms. Unlike localisée dans la région du nerf trijumeau, mais il existe trigeminal neuralgia, the condition encompasses a wider un certain nombre de rapports de cas qui décrivent des distribution that does not conform or relate to a specific symptômes bilatéraux. Contrairement à la névralgie dermatome. In addition, the pain is typically continuous, faciale, l’affection englobe une distribution plus with no periods of remission and there are no signs or large qui n’est pas conforme ou liée à un dermatome symptoms suggestive of autonomic involvement. Reports précis. De plus, la douleur est généralement continue, documenting the response to various conservative sans période de rémission et il n’y a aucun signe ou treatments for persistent idiopathic facial pain have been symptôme suggérant une atteinte du système nerveux widely variable likely due to the heterogeneity of the autonome. Les rapports documentant la réponse à condition. Four cases of persistent idiopathic facial pain divers traitements conservateurs utilisés pour la douleur faciale idiopathique persistante ont été très variables, probablement en raison de l’hétérogénéité de l’affection. Quatre cas de douleur faciale idiopathique persistante due à un soupçon de neuropathie du nerf trijumeau et 1 Private practice, Oakville, Ontario 2 Canadian Memorial Chiropractic College Corresponding author: Brad Muir, 6100 Leslie Street, Toronto, ON M2H 3J1 Tel: (416) 482-2340 E-mail: [email protected] © JCCA 2019 The authors have no disclaimers, competing interests, or sources of support or funding to report in the preparation of this manuscript. The involved patients provided consent for case publication. 126 J Can Chiropr Assoc 2019; 63(2) N Moser, B Muir due to suspected trigeminal nerve neuropathy and their leur prise en charge sont présentés. Dans les quatre management are presented. A specific form of targeted, cas décrits, une forme spécifique de thérapie par manual, instrument-assisted, intra-oral vibration therapy vibration ciblée, intraorale, manuelle, appuyée par des appeared to provide relief in the four cases described. instruments, a semblé apporter un soulagement. (JACC. 2019;63(2):126-138) (JCCA. 2019;63(2):126-138) mots clés : nerf trijumeau1, douleur faciale, key words : trigeminal nerve1, facial pain, chiropratique, thérapie par vibration, douleur faciale chiropractic, vibration therapy, atypical facial pain atypique. Introduction with PPTTN defined as “spontaneous or touch evoked Lifetime prevalence of non-specific facial pain is esti- pain predominantly affecting the receptive field of one mated at 26%.1 The condition presents as a diagnostic or more divisions of the trigeminal nerve. The duration challenge for even the most experienced clinicians due to ranges widely from episodic (minutes to days) and may its multifaceted presentation and etiology. also be constant. The pain tends to spread with time and Persistent idiopathic facial pain (PIFP) is typically is mostly unilateral without crossing the midline.”11 (pg 50) described as deep, poorly localized pain in the territory They also suggest that the pain develops within three of the trigeminal nerve. It is most often unilateral, how- months of an identifiable trauma and lasts for more than 3 ever approximately one-third of patients develop bilat- months.11 eral symptoms.2 PIFP often spreads to a wider area, not The purpose of this article is to familiarize the clin- conforming to a specific dermatome. In addition, the pain ician with the clinical presentation, diagnosis, etiology is typically continuous with no periods of remission and and management of PIFP secondary to trigeminal nerve there are no signs or symptoms of autonomic involve- neuropathy as currently understood. Four cases are pre- ment.2 Recent evidence supports the notion that PIFP may sented to illustrate the features of the disorder. itself be a neuropathic pain condition.2, 3-7 Friedman et al.8 demonstrated consistent maxillary alveolar tenderness Case presentations (MAT) in the distribution of the maxillary nerve on the in- volved side of the face in patients diagnosed with atypical Case 1 facial pain. The authors state that this tenderness is often A 28-year old female employed as a full-time medic- incorrectly attributed to lateral pterygoid hypertonicity or al receptionist presented to a chiropractic clinic with a spasm, despite the inaccessibility of this muscle to digital 1.5-year history of bilateral facial pain located over the palpation.8 They found intra-oral tenderness in the max- temporal and temporomandibular joint regions. The onset illary molar periapical area in over 90% of asymptomatic was attributed having a “nerve struck” while undergoing migraine patients, with laterality and degree of tenderness extensive dental work for caries. Subsequently, a root closely related to headache symptoms.9 Significant ipsi- canal and a dental crown were required. The pain over the lateral tenderness and increased local temperature was temporomandibular region was described as an intermit- found in the maxillary molar periapical area on palpation tent dull ache. The temporal headache pain was described bilaterally during unilateral migraine, tension type head- as a constant squeezing and pressure sensation occurring aches, and facial pain.10 weekly. Both painful areas were rated at seven out of 10 Neuropathic pain secondary to trigeminal nerve injury on the numerical rating scale (NRS). The major aggravat- has also been poorly defined in the literature.11 Benoliel et ing factor was clenching. The pain was worse in the mor- al.11 has proposed a modified diagnostic criteria for per- ning and after work. The patient denied any lancinating, ipheral painful traumatic trigeminal neuropathy (PPTTN) burning or paroxysmal sensations in the face and denied J Can Chiropr Assoc 2019; 63(2) 127 Suspected trigeminal nerve neuropathy causing persistent idiopathic facial pain: a report of four cases Figure 1. Location of the infraorbital and supraorbital foramen. The infraorbital foramen is the exit point of the infraorbital nerve from the skull (red dot indicated by the blue arrow). By pinching the skin over the eyebrow Figure 2. directly over the pupillary line (with midline gaze), the Proposed trigeminal nerve tension test. Palpating the supraorbital foramen (or notch) can be easily palpated supraorbital nerve just superior to the supraorbital and subsequently the supraorbital nerve (indicated by foramen (or notch) (red arrow) the patient is asked to the red arrow). The location of the pupil (white dot) is open their mouth. An increase in tension and/or pain indicated by the grey arrow. would indicate a positive test. any associated nausea, vomiting, photophobia, phono- of the right temporomandibular joint (TMJ). There was phobia and auras. At baseline, her Jaw Functional Limit- moderate tenderness over the temporalis, masseter and ation Scale score was zero, indicating no limitations. The medial pterygoid muscles bilaterally. Palpation over the Jaw Functional Limitation Scale has demonstrated good infraorbital and supraorbital foramen, the exit point from reliability and construct validity in assessing limitations the skull of the infra (See Figure 1) and supraorbital (See in mastication, jaw mobility and verbal and emotional ex- Figure 1) nerves, respectively, elicited tenderness, which pression.12 increased on mouth opening (See Figure 2) recreating a On examination, the patient weighed 54 kg and stood lesser version of her temporal headache pain. Intra-oral 163 cm tall. Active and passive ranges of motion of the palpation along the superior mucobuccal fold in the area cervical spine were full and pain free. Orthopaedic testing of the superior alveolar nerve and inferior mucobuccal of the cervical spine, inclusive of Spurling’s and Jack- fold at the exit and distribution of the mental nerve, the son’s, was unremarkable. The cervical paraspinal muscu- terminal branch of the inferior alveolar nerve, was locally lature was diffusely tender, however no pain referred to tender bilaterally reproducing her presenting symptoms. the areas of the chief complaint. A rightward C-shaped Examination of other cranial nerves and peripheral nerv- shift of the mandible was visualized during jaw open- ous system was within normal limits. ing but no obvious clicking was noted. A palpable clunk An MRI, ordered by her family medical doctor, of both was noted bilaterally upon closing the mouth. No lock- temporomandibular joints
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